Behavioural and physiological indicators of anxiety reflect shared and distinct profiles across individuals with neurogenetic syndromes

Hayley Crawford, Chris Oliver, Laura Groves, Louise Bradley, Kayla Smith, Abigail Hogan, Derek Renshaw, Jane Waite, Jane Roberts

Research output: Contribution to journalArticlepeer-review


Anxiety is heightened in individuals with intellectual disability, particularly in those with specific neurogenetic syndromes. Assessment of anxiety for these individuals is hampered by a lack of appropriate measures that cater for communication impairment, differences in presentation, and overlapping features with co-occurring conditions. Here, we adopt a multi-method approach to identify fine-grained behavioural and physiological (via salivary cortisol) responses to anxiety presses in people with fragile X (FXS; n = 27; M age = 20.11 years; range 6.32 - 47.04 years) and Cornelia de Lange syndromes (CdLS; n = 27; M age = 18.42 years; range 4.28 - 41.08 years), two neurogenetic groups at high risk for anxiety, compared to neurotypical children (NT; n = 21; M age = 5.97 years; range 4.34 - 7.30 years). Results indicate that physical avoidance of feared stimuli and proximity seeking to a familiar adult are prominent behavioural indicators of anxiety/stress in FXS and CdLS. Heightened pervasive physiological arousal was identified in these groups via salivary cortisol. An association between autistic characteristics and anxiety was evident in the FXS group but not in the CdLS group pointing to syndrome-specific nuances in the association between anxiety and autism. This study furthers understanding of the behavioural and physiological presentation of anxiety in individuals with intellectual disability and progresses theoretical developments regarding the development and maintenance of anxiety at the intersection of autism.

Original languageEnglish
Article number115278
Number of pages9
JournalPsychiatry Research
Early online date30 May 2023
Publication statusPublished - Aug 2023

Bibliographical note

Copyright © 2023 The Author(s). Published by Elsevier B.V. This is an open access article under the CC BY license (

Funding: This work was supported by Baily-Thomas Charitable Fund; National Institute for Health Research (NIHR) Applied Research Collaboration (ARC) West Midlands; Cornelia de Lange Syndrome Foundation UK and Ireland; and Cerebra. The views expressed are those of the author(s) and not necessarily those of the NIHR or the Department of Health and Social Care.


  • Fragile X syndrome
  • Cornelia de Lange syndrome
  • Cortisol


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